Blepharospasm in Japan: A Clinical Observational Study From a Large Referral Hospital in Tokyo

Wakakura, Masato; Yamagami, Akiko; Iwasa, Mayumi

doi:10.1080/01658107.2017.1409770

Cited by 10 publications

(20 citation statements)

References 26 publications

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“…Twenty-one reported median age at onset in 5–6th decade, with an overall weighted average of 56 ± 6 years ( Figure 2B ). Six reported diagnosis was significantly delayed; two of these revealed that only about half of the patients received the correct diagnosis within one year [ 4 , 10 ]. The median time from onset to diagnosis was 2 years in one study [ 22 ] and the weighted mean from three other studies was 46 ± 68 months [ 7 , 11 , 21 ].…”

Section: Resultsmentioning

confidence: 99%

“…However, its varied presentations are often not appreciated, and it is probably under-diagnosed. Several reports describe frequent misdiagnoses [ 5 , 7 , 10 ]. Initial diagnostic accuracy was only 19% in one study [ 10 ] and 10% in another [ 7 ].…”

Section: Introductionmentioning

confidence: 99%

“…Several reports describe frequent misdiagnoses [ 5 , 7 , 10 ]. Initial diagnostic accuracy was only 19% in one study [ 10 ] and 10% in another [ 7 ]. The most common misdiagnoses included eye strain or eye irritation from dryness, conjunctivitis, or keratitis.…”

Section: Introductionmentioning

confidence: 99%

“…The most common misdiagnoses included eye strain or eye irritation from dryness, conjunctivitis, or keratitis. These diagnostic errors contribute to delays between onset and diagnosis that may span many years [ 4 , 7 , 10 , 11 , 19 - 22 ]. Although improved awareness in recent years has shortened these delays, more than half of all affected individuals typically require more than a year to reach a diagnosis [ 21 ].…”

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Clinical Features and Evolution of Blepharospasm: A Multicenter International Cohort and Systematic Literature Review

Scorr¹,

Cho²,

Kilic‐Berkmen³

et al. 2022

Dystonia

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Objective: Blepharospasm is a type of dystonia where the diagnosis is often delayed because its varied clinical manifestations are not well recognized. The purpose of this study was to provide a comprehensive picture of its clinical features including presenting features, motor features, and non-motor features.Methods: This was a two-part study. The first part involved a systematic literature review that summarized clinical features for 10,324 cases taken from 41 prior reports. The second part involved a summary of clinical features for 884 cases enrolled in a large multicenter cohort collected by the Dystonia Coalition investigators, along with an analysis of the factors that contribute to the spread of dystonia beyond the periocular region.Results: For cases in the literature and the Dystonia Coalition, blepharospasm emerged in the 50s and was more frequent in women. Many presented with non-specific motor symptoms such as increased blinking (51.9%) or non-motor sensory features such as eye soreness or pain (38.7%), photophobia (35.5%), or dry eyes (10.7%). Non-motor psychiatric features were also common including anxiety disorders (34–40%) and depression (21–24%). Among cases presenting with blepharospasm in the Dystonia Coalition cohort, 61% experienced spread of dystonia to other regions, most commonly the oromandibular region and neck. Features associated with spread included severity of blepharospasm, family history of dystonia, depression, and anxiety.Conclusions: This study provides a comprehensive summary of motor and non-motor features of blepharospasm, along with novel insights into factors that may be responsible for its poor diagnostic recognition and natural history.

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Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Clinical Features and Evolution of Blepharospasm: A Multicenter International Cohort and Systematic Literature Review

Scorr¹,

Cho²,

Kilic‐Berkmen³

et al. 2022

Dystonia

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“…There have been several reports of this symptom since ancient times 20 - 24 . In a recent study, Wakakura et al 25 conducted a retrospective observational study involving a large population of patients from a single institution, and reported the need to remain aware of “benzodiazepine-induced ophthalmopathy” when prescribing etizolam, BZ drugs, and zolpidem (= Z-drug). Therefore, when switching from BZ to Z-drugs, it is essential to pay close attention to “benzodiazepine-induced ophthalmopathy” as Ocular signs and symptoms NEC.…”

Section: Discussionmentioning

confidence: 99%

Differences in detected safety signals between benzodiazepines and non-benzodiazepine hypnotics: pharmacovigilance study using a spontaneous reporting system

et al. 2021

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A Prediction Rule for the Dystonic Spread of Blepharospasm: A 4‐Year Prospective Cohort Study

Xing,

Hu,

Teng

et al. 2024

Annals of Neurology

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ObjectiveBlepharospasm (BSP), focal dystonia with the highest risk of spread, lacks clear understanding of early spreading risk factors and objective prognostic indicators. We aimed to identify these risk factors through clinical and electrophysiological assessments, and to establish a predictive model for dystonic spread in BSP.MethodsWe prospectively followed BSP patients for 4 years, collecting data on dystonic spread, and conducting electrophysiological evaluations. The blink reflex, masseter inhibitory reflex, and trigeminal somatosensory evoked potential were assessed. Univariable and multivariable Cox proportional hazard regression models were used to assess clinical characteristics associated with BSP dystonic spread. A predictive model was constructed using a nomogram, and performance of the model was evaluated using the area under the receiver operating characteristic curve.ResultsA total of 136 enrolled participants (mean age 56.34 years) completed a 4‐year follow‐up. Among them, 62 patients (45.6%) showed spread to other body regions. Multivariable Cox regression analysis showed that a high Hamilton Anxiety Scale score (hazard ratio 1.19, 95% confidence interval 1.13–1.25, p < 0.001), prolonged trigeminal somatosensory evoked potential mandibular branch P1‐N2 peak interval (hazard ratio 1.11, 95% confidence interval 1.02–1.21, p = 0.017), and elevated trigeminal somatosensory evoked potential mandibular branch P1‐N2 peak amplitude (hazard ratio 1.26, 95% confidence interval 1.12–1.41, p < 0.001) were independent risk factors for BSP dystonic spread within 4 years. Combining these factors, the predictive models demonstrated excellent discriminative ability, with the receiver operating characteristic curve score being 0.797, 0.790, 0.847, and 0.820 at 1, 2, 3 and 4 years after enrollment, respectively.InterpretationWe established a predictive model with significant value for anticipating dystonic spread in BSP, offering crucial evidence. These findings contribute essential insights into the early clinical identification of the development and evolution of BSP diseases. ANN NEUROL 2024

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Blepharospasm in Japan: A Clinical Observational Study From a Large Referral Hospital in Tokyo

Cited by 10 publications

References 26 publications

Clinical Features and Evolution of Blepharospasm: A Multicenter International Cohort and Systematic Literature Review

Clinical Features and Evolution of Blepharospasm: A Multicenter International Cohort and Systematic Literature Review

Differences in detected safety signals between benzodiazepines and non-benzodiazepine hypnotics: pharmacovigilance study using a spontaneous reporting system

A Prediction Rule for the Dystonic Spread of Blepharospasm: A 4‐Year Prospective Cohort Study

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